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@ARTICLE{Brenner:283019,
author = {Brenner, Juliette and Bastiaansen, Anna E. M. and Guasp,
Mar and Muñiz-Castrillo, Sergio and Iizuka, Takahiro and de
Bruijn, Marienke A. A. M. and Muñoz-Lopetegi, Amaia and
Martínez-Hernández, Eugenia and Picard, Géraldine and
Vogrig, Alberto and Millot, Mathilde and Finke, Carsten and
Geis, Christian and Lewerenz, Jan and Melzer, Nico and
Prüss, Harald and Räuber, Saskia and Ringelstein, Marius
and Rostàsy, Kevin and Sühs, Kurt-Wolfram and Thaler,
Franziska S. and Wandinger, Klaus-Peter and Wurdack,
Katharina and Crijnen, Yvette S. and Kerstens, Jeroen and
van Steenhoven, Robin W. and Veenbergen, Sharon and
Schreurs, Marco W. J. and van den Berg, Robert and Volovici,
Victor and Neuteboom, Rinze F. and de Vries, Juna M. and
Sillevis Smitt, Peter A. E. and Nagtzaam, Mariska M. P. and
Franken, Suzanne C. and Ratuszny, Dominica and Menge, Til
and Bertolini, Annikki and Bien, Christian and Berger,
Robert and Tauber, Simone and Angstwurm, Klemens and
Seifert-Held, Thomas and Kraft, Andrea and Klausewitz,
Jaqueline and Ayzenberg, Ilya and Eisenhut, Katharina and
Roessling, Rosa and Heiden, Martha and Kümpfel, Tania and
Dalmau, Josep and Leypoldt, Frank and Honnorat, Jérôme and
Titulaer, Maarten J.},
title = {{D}evelopment and validation of the {NEOS}2 score for
prediction of long-term outcomes and improvement after
first-line immunotherapy in patients with anti-{NMDAR}
encephalitis: an international cohort study},
journal = {The lancet / Regional health. Europe},
volume = {62},
issn = {2666-7762},
address = {[Amsterdam]},
publisher = {Elsevier},
reportid = {DZNE-2025-01431},
pages = {101562},
year = {2026},
note = {Funding: This study was funded by Dioraphte (charity;
project 2001 0403).},
abstract = {Background: Anti-N-methyl-D-aspartate receptor (anti-NMDAR)
encephalitis is a severe disease that primarily affects
young people and can improve with adequate treatment. We
aimed to refine the anti-NMDAR Encephalitis One-year
functional Status (NEOS) score by developing NEOS2, an
updated model using readily available data at the time of
diagnosis. We assessed the predictive value of the
NEOS2-score for (1) improvement following first-line
treatment, (2) functional outcome at one-year follow-up, and
(3) resumption of school or work within three years.
Methods: In this international (France, Germany, Japan, the
Netherlands and Spain) cohort study in patients with a
definite anti-NMDAR encephalitis diagnosis (according to the
clinical criteria plus antibody testing in CSF), we
performed logistic regression analyses to develop and
validate multivariable models to predict -based upon
variables available at diagnosis- short (ΔmRS two weeks
after first-line treatment), middle (modified Rankin Scale
[mRS] at one year), and long-term (return to school or work
within three years) outcomes. We included clinical variables
and biomarkers available at diagnosis. Findings: We included
702 patients (mean age 23 years, $95\%-CI$ 2–69; $79\%$
female, $21\%$ male) diagnosed between the discovery of the
disease in 2007 and 2022. Most patients $(96\%;$ 672/702)
had received first-line immunotherapy, and $38\%$ (233/615)
showed improvement within two weeks. One year after
diagnosis, $80\%$ (517/644) had a favourable functional
outcome (mRS≤2). At three years, $73\%$ (203/278) had
resumed work/school. In multivariable analysis, higher age
(odds ratio [OR] 0·35, $95\%-CI$ 0·29–0·43, p <
0·0001), treatment delay (OR 0·49, $95\%-CI$
0·41–0·58, p < 0·0001), movement disorders (OR 0·32,
$95\%-CI$ 0·24–0·41, p < 0·0001), ICU-requirement (OR
0·34, $95\%-CI$ 0·26–0·44, p < 0·0001) and increased
CSF leucocyte count (OR 0·65, $95\%-CI$ 0·60–0·71, p <
0·0001) independently predicted poorer outcomes (NEOS2,
accuracy AUC $80\%,$ $95\%-CI$ $75–86\%).$ The same
variables, excluding age, were relevant in predicting
improvement following first-line immunotherapy (NEOS2-T AUC
$81–84\%,$ $95\%-CI$ $77–86\%).$ Return-to-work or
-school served as a useful measure of longer-term outcomes,
predicted with equal accuracy as one-year functional outcome
(NEOS2-W AUC $80\%,$ $95\%-CI$ $75–85\%).$ The
NEOS2-score, applied as an ordinal measure, enabled nuanced
predictions of outcome probabilities across the score
spectrum, ranging from a high $(80\%;$ n = 20/25) likelihood
of improving after first-line immunotherapy and achieving a
good outcome $(100\%;$ n = 32/32) to a high risk of
first-line treatment failure $(97\%;$ n = 77/79) and no
return to school/work $(94\%;$ n = 15/16). Interpretation:
The NEOS2-score, readily available at diagnosis and easy to
apply, can identify patients with either a favourable or
poor prognosis, and those who may benefit from early
intensified treatment. The value of the NEOS2-score for
guiding treatment decisions and as a stratification tool in
studies on optimal treatment regimens, should be confirmed
in further prospective studies.},
cin = {AG Prüß},
ddc = {610},
cid = {I:(DE-2719)1810003},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)16},
doi = {10.1016/j.lanepe.2025.101562},
url = {https://pub.dzne.de/record/283019},
}